Quality of care for people with differences of sex development (DSD) in Germany

Maike Schnoor; Andreas Heidenreich; Martina Jürgensen; Ulla Döhnert; Olaf Hiort; Alexander Katalinic

doi:10.1186/s13023-024-03467-5

Quality of care for people with differences of sex development (DSD) in Germany

Maike Schnoor, Andreas Heidenreich, Martina Jürgensen, Ulla Döhnert, Olaf Hiort, Alexander Katalinic

Abstract

Background: People with "Differences of Sex Development" (DSD) require comprehensive, specialised, and individualised medical and psychological care. This is often perceived as inadequate by those affected. Therefore, the German Federal Ministry of Health funded the project DSDCare which aimed to improve the quality of care for people with DSD over the lifespan in Germany. Method: Indicators of structural, process, and outcome quality were defined, which are used to evaluate the quality of care. The indicators of structural quality are collected once a year from ten participating centres. Based on the "Open Source Registry System for Rare Diseases (OSSE)", a DSD-specific registry (DSDReg) was developed, in which patient-related care data are recorded in order to be able to assess the process and outcome quality. Furthermore, patient-reported outcomes are collected directly from the patients by means of questionnaires. The data are reported back to the participating centres in an annual benchmarking. Results: Twenty-five indicators of structural quality were defined, twelve indicators of process quality and ten of outcome quality. A total of 477 patients were registered in DSDReg in the period from May 2021 till October 2022. The mean age is 16 years; the most common diagnosis groups are 46,XY DSD (34.8%), followed by 46,XX DSD (33.3%) and chromosomal DSD (27.5%). Patient numbers vary across centres from N = 10 to N = 131. Questionnaires are available from 316 (66.2%) affected individuals, including 122 from adults, 120 from children or adolescents with DSD, and 191 from parents. Preliminary results show heterogeneity between centres in both data quality and quality of care. Conclusions: The DSDReg is well established in the DSDCare project as a quality assurance tool with continuously increasing recruitment figures. The implemented quality indicators are applicable, enable a comparison between the participating centres and will foreseeably lead to an improvement of the care of patients with DSD. A long-term continuation of the registry after the end of the initial study period is therefore indicated.

Original language	English
Article number	460
Journal	Orphanet Journal of Rare Diseases
Volume	19
Issue number	1
Pages (from-to)	460
ISSN	1750-1172
DOIs	https://doi.org/10.1186/s13023-024-03467-5
Publication status	Published - 12.2024

Funding

Funders	Funder number
Bundesministerium für Gesundheit	2519FSB503

Research Areas and Centers

Research Area: Center for Population Medicine and Public Health (ZBV)

DFG Research Classification Scheme

2.22-17 Endocrinology, Diabetology, Metabolism
2.22-20 Pediatric and Adolescent Medicine
2.22-02 Public Health, Healthcare Research, Social and Occupational Medicine
1.22-03 Developmental and Educational Psychology
1.22-05 Personality Psychology, Clinical and Medical Psychology, Methodology

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

Access to Document

10.1186/s13023-024-03467-5

Cite this

@article{7031ef68a50d4541975ece7ff2e5b92b,

title = "Quality of care for people with differences of sex development (DSD) in Germany",

abstract = "Background: People with {"}Differences of Sex Development{"} (DSD) require comprehensive, specialised, and individualised medical and psychological care. This is often perceived as inadequate by those affected. Therefore, the German Federal Ministry of Health funded the project DSDCare which aimed to improve the quality of care for people with DSD over the lifespan in Germany. Method: Indicators of structural, process, and outcome quality were defined, which are used to evaluate the quality of care. The indicators of structural quality are collected once a year from ten participating centres. Based on the {"}Open Source Registry System for Rare Diseases (OSSE){"}, a DSD-specific registry (DSDReg) was developed, in which patient-related care data are recorded in order to be able to assess the process and outcome quality. Furthermore, patient-reported outcomes are collected directly from the patients by means of questionnaires. The data are reported back to the participating centres in an annual benchmarking. Results: Twenty-five indicators of structural quality were defined, twelve indicators of process quality and ten of outcome quality. A total of 477 patients were registered in DSDReg in the period from May 2021 till October 2022. The mean age is 16 years; the most common diagnosis groups are 46,XY DSD (34.8\%), followed by 46,XX DSD (33.3\%) and chromosomal DSD (27.5\%). Patient numbers vary across centres from N = 10 to N = 131. Questionnaires are available from 316 (66.2\%) affected individuals, including 122 from adults, 120 from children or adolescents with DSD, and 191 from parents. Preliminary results show heterogeneity between centres in both data quality and quality of care. Conclusions: The DSDReg is well established in the DSDCare project as a quality assurance tool with continuously increasing recruitment figures. The implemented quality indicators are applicable, enable a comparison between the participating centres and will foreseeably lead to an improvement of the care of patients with DSD. A long-term continuation of the registry after the end of the initial study period is therefore indicated.",

author = "Maike Schnoor and Andreas Heidenreich and Martina J{\"u}rgensen and Ulla D{\"o}hnert and Olaf Hiort and Alexander Katalinic",

note = "Publisher Copyright: {\textcopyright} The Author(s) 2024.",

year = "2024",

month = dec,

doi = "10.1186/s13023-024-03467-5",

language = "English",

volume = "19",

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T1 - Quality of care for people with differences of sex development (DSD) in Germany

AU - Schnoor, Maike

AU - Heidenreich, Andreas

AU - Jürgensen, Martina

AU - Döhnert, Ulla

AU - Hiort, Olaf

AU - Katalinic, Alexander

PY - 2024/12

Y1 - 2024/12

N2 - Background: People with "Differences of Sex Development" (DSD) require comprehensive, specialised, and individualised medical and psychological care. This is often perceived as inadequate by those affected. Therefore, the German Federal Ministry of Health funded the project DSDCare which aimed to improve the quality of care for people with DSD over the lifespan in Germany. Method: Indicators of structural, process, and outcome quality were defined, which are used to evaluate the quality of care. The indicators of structural quality are collected once a year from ten participating centres. Based on the "Open Source Registry System for Rare Diseases (OSSE)", a DSD-specific registry (DSDReg) was developed, in which patient-related care data are recorded in order to be able to assess the process and outcome quality. Furthermore, patient-reported outcomes are collected directly from the patients by means of questionnaires. The data are reported back to the participating centres in an annual benchmarking. Results: Twenty-five indicators of structural quality were defined, twelve indicators of process quality and ten of outcome quality. A total of 477 patients were registered in DSDReg in the period from May 2021 till October 2022. The mean age is 16 years; the most common diagnosis groups are 46,XY DSD (34.8%), followed by 46,XX DSD (33.3%) and chromosomal DSD (27.5%). Patient numbers vary across centres from N = 10 to N = 131. Questionnaires are available from 316 (66.2%) affected individuals, including 122 from adults, 120 from children or adolescents with DSD, and 191 from parents. Preliminary results show heterogeneity between centres in both data quality and quality of care. Conclusions: The DSDReg is well established in the DSDCare project as a quality assurance tool with continuously increasing recruitment figures. The implemented quality indicators are applicable, enable a comparison between the participating centres and will foreseeably lead to an improvement of the care of patients with DSD. A long-term continuation of the registry after the end of the initial study period is therefore indicated.

AB - Background: People with "Differences of Sex Development" (DSD) require comprehensive, specialised, and individualised medical and psychological care. This is often perceived as inadequate by those affected. Therefore, the German Federal Ministry of Health funded the project DSDCare which aimed to improve the quality of care for people with DSD over the lifespan in Germany. Method: Indicators of structural, process, and outcome quality were defined, which are used to evaluate the quality of care. The indicators of structural quality are collected once a year from ten participating centres. Based on the "Open Source Registry System for Rare Diseases (OSSE)", a DSD-specific registry (DSDReg) was developed, in which patient-related care data are recorded in order to be able to assess the process and outcome quality. Furthermore, patient-reported outcomes are collected directly from the patients by means of questionnaires. The data are reported back to the participating centres in an annual benchmarking. Results: Twenty-five indicators of structural quality were defined, twelve indicators of process quality and ten of outcome quality. A total of 477 patients were registered in DSDReg in the period from May 2021 till October 2022. The mean age is 16 years; the most common diagnosis groups are 46,XY DSD (34.8%), followed by 46,XX DSD (33.3%) and chromosomal DSD (27.5%). Patient numbers vary across centres from N = 10 to N = 131. Questionnaires are available from 316 (66.2%) affected individuals, including 122 from adults, 120 from children or adolescents with DSD, and 191 from parents. Preliminary results show heterogeneity between centres in both data quality and quality of care. Conclusions: The DSDReg is well established in the DSDCare project as a quality assurance tool with continuously increasing recruitment figures. The implemented quality indicators are applicable, enable a comparison between the participating centres and will foreseeably lead to an improvement of the care of patients with DSD. A long-term continuation of the registry after the end of the initial study period is therefore indicated.

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Quality of care for people with differences of sex development (DSD) in Germany

Abstract

Funding

Research Areas and Centers

DFG Research Classification Scheme

UN SDGs

Access to Document

Other files and links

Fingerprint

SFB 1665: Sexdiversity - Determinants, meanings and implications of sex diversity in sociocultural, medical and biological landscapes

Cite this

Quality of care for people with differences of sex development (DSD) in Germany

Abstract

Funding

Research Areas and Centers

DFG Research Classification Scheme

UN SDGs

Access to Document

Other files and links

Fingerprint

Projects

SFB 1665: Sexdiversity - Determinants, meanings and implications of sex diversity in sociocultural, medical and biological landscapes

Cite this